Abstract
We report the first case of vertebral aspergillosis in a child with a primary defect in monocyte killing, an extremely rare immunodeficiency The diagnosis of defective monocyte killing was made by an in vitro assay that showed normal killing of Staphylococcus aureus by the patient's neutrophils but impaired killing by his monocytes. Importantly, the extensive granulomatous infection that involved the vertebral column, posterior mediastinum, pleura, and lung was not responsive to aggressive treatment with a combination of liposomal amphotericin B. intralesional amphotericin B. itraconazole, and granulocyte transfusions. Dramatic clinical and radiological improvement was only seen after the addition of granulocyte macrophage-colony stimulating factor (GM-CSF) to his treatment regimen. The use of GM-CSF in the treatment of invasive aspergillosis in immunocompromised patients requires further evaluation.
MeSH terms
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Amphotericin B / administration & dosage
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Amphotericin B / therapeutic use
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Antifungal Agents / administration & dosage
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Antifungal Agents / therapeutic use
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Aspergillosis / diagnosis*
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Aspergillosis / drug therapy
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Aspergillus / isolation & purification
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Biopsy, Needle
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Child, Preschool
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Flucytosine / therapeutic use
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Granulocyte-Macrophage Colony-Stimulating Factor / administration & dosage
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Granulocyte-Macrophage Colony-Stimulating Factor / therapeutic use*
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Histocytochemistry
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Humans
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Immunocompromised Host*
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Itraconazole / therapeutic use
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Leukocyte Count
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Magnetic Resonance Imaging
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Male
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Monocytes / immunology*
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Monocytes / pathology
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Osteomyelitis / diagnosis*
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Osteomyelitis / drug therapy
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Radiography, Thoracic
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Spinal Diseases / diagnosis*
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Spinal Diseases / drug therapy
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Spine / pathology
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Tomography, X-Ray Computed
Substances
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Antifungal Agents
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Itraconazole
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Amphotericin B
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Granulocyte-Macrophage Colony-Stimulating Factor
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Flucytosine